Multiple intracranial and spinal metastases from a ... - Springer Link

Wang et al. World Journal of Surgical Oncology 2014, 12:380 http://www.wjso.com/content/12/1/380

WORLD JOURNAL OF SURGICAL ONCOLOGY

CASE REPORT

Open Access

Multiple intracranial and spinal metastases from a nonfunctioning pituitary adenoma following multiple surgeries: an illustrative case with 16 years of follow-up Jun Wang, Er-meng Ma, Peng-fei Wu, Bo Qiu and Yun-jie Wang*

Abstract Pituitary adenomas are the third most common primary intracranial tumor; however, those with postoperative metastases are very rare and are classically considered as pituitary carcinomas. The field of neurosurgery has struggled with diagnosing and treating these unusual lesions. In this report, we retrospectively analyze the clinical features, imaging findings, pathological characteristics and prognosis of one patient with non-hormone-secreting pituitary adenoma who had multiple intracranial and spinal metastases and underwent four surgeries in a 16-year follow-up period. In addition, on the basis of the existing literature, we explore the underlying mechanisms of, as well as the preventive and therapeutic strategies used to treat, pituitary carcinomas and postoperative metastasis of pituitary tumors. Keywords: Metastasis, Microsurgery, Pituitary adenoma

Background Pituitary adenomas are very common and rank third in terms of incidence among all primary intracranial tumors. However, primary pituitary adenomas with intracranial or extracranial metastases are very rare and are commonly defined as malignant pituitary tumors or pituitary carcinomas [1-7]. The field of neurosurgery has struggled with the diagnosis and treatment of these unusual lesions. In general, the prognosis of this disease is not optimistic. Therefore, it demands an early diagnosis and effective therapies. In this report, we present the case of one patient with non-hormone-secreting pituitary adenoma who had multiple intracranial and spinal metastases and underwent four surgeries in a 16-year period. In addition, on the basis of a literature review, we discuss the diagnostic, preventive and therapeutic strategies for malignant pituitary adenoma and postoperative metastasis of pituitary tumors.

Case presentation History

A 40-year-old man was hospitalized (the fourth admission) with a major complaint of declining visual acuity during the preceding 6 months. He underwent his first craniotomy in 1998 for removal of a large pituitary adenoma (Figure 1). The final pathological diagnosis was benign pituitary adenoma. In 2008 and 2010, he was readmitted to our hospital with hearing loss in the left ear and numbness and weakness in his limbs, and the second and third surgeries were performed for the removal of lesions at different sites, respectively (Figure 1). Generally, the histological examinations of all surgical specimens obtained from the second and third operations showed typical benign pituitary adenomas. After the third operation, whole-brain and spinal cord radiation therapy was recommended because the tumor was then clinically diagnosed as a malignant lesion due to its behavior of recurring metastases. Unfortunately, because of the possible side effects, the patient refused to

* Correspondence: [email protected] Department of Neurosurgery, The First Hospital of China Medical University, No. 155, Nanjing North Street, Heping Ward, Shenyang 110001, China © 2014 Wang et al.; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.


Page 2 of 7

Figure 1 Pre- and postoperative magnetic resonance imaging scans from the first three surgeries. (A to D) Preoperative magnetic resonance imaging (MRI) scans obtained in December 1998 show a typical pituitary tumor. (E to H) and (F to K) Postoperative MRI scans show complete resection of the tumor (first surgery). (I to M) MRI scans taken in May 2008 show a lesion in the left cerebellopontine angle area. (N to P) Postoperative MRI scans show complete resection of the lesion (second surgery). (Q and R) MRI scans obtained in October 2010 show a lesion at the C4-C5 spinal level. (S to U) Postoperative MRI scans show complete resection of the lesion (third surgery).

undergo the recommended treatment. On physical examination at the fourth admission, the patient was conscious and could answer questions correctly. Mild bilateral papilledema was detected. The patient had bitemporal hemianopsia and declining bilateral visual acuity (left: 4.8, right: 5.0). He had no other neurological deficit.

Laboratory and neuroimaging examinations

The laboratory findings in May 2013 (at the time of the patient’s fourth admission) showed that his serum hormone levels, including prolactin (PRL) (225.0 mIU/L; reference ranges, 53.0 to 360.0 mIU/L), adrenocorticotropic hormone (ACTH) (47.43 pg/ml; reference range, 7.20 to 63.30 pg/ml), human growth hormone (hGH)


(0.22 mIU/L; reference range, 0.16 to 2.60 mIU/L), insulin-like growth factor 1 (1.01 U/ml; reference range, 0.5 to 2.0 U/ml) and thyroid-stimulating hormone (TSH) (2.243 mIU/L; reference range, 0.350 to 4.940 mIU/L), were all normal. His visual field examination indicated bitemporal hemianopsia. Contrast-enhanced magnetic resonance imaging (MRI) of the patient’s brain indicated the presence of three intracranial lesions, with their locations being in the sella turcica (maximum diameter, 2.0 × 1.2 × 2.2 cm), the right frontal lobe (maximum diameter, 0.5 × 0.4 × 0.3 cm) and the anterior cerebral falx (maximum diameter, 0.6 × 0.5 × 0.3 cm), respectively. All lesions demonstrated long T1 and T2 heterogeneous signals and high signal intensity on fluid-attenuated inversion recovery images with significant enhancement (Figures 2 and 3). Surgical procedures

In the patient’s fourth surgery, the right subfrontal approach of craniotomy was adopted, and the incision was made at the same site used in the first surgery in 1998. After we expanded the bone window to the middle line and attempted to cut the dura mater open, we observed the dura mater to be tightly adhered to the right frontal cortex, which was then carefully dissociated under a microscope. During the dissociation process, we first exposed and removed the lesion in the right frontal lobe. The lesion was gray and soft, with a base connected to the dura mater. The lesion was immediately sent for intraoperative pathological examination, and the diagnosis was neuroendocrine neoplasm. Subsequently, we dissociated and removed the tumor in the sellar region. We then exposed and resected the tumor at the cerebral falx using an interhemispheric fissure approach. After the surgery, the patient had a satisfactory recovery of visual field, but experienced transient diabetes insipidus and hypopituitarism, from which he gradually recovered after symptomatic treatment. No other operation-related complications

Page 3 of 7

occurred. The postoperative MRI demonstrated complete removal of three lesions. Whole-brain and spine radiotherapy in combination with chemotherapy were suggested to the patient, but he rejected this suggestion. Postoperative pathology

Pathological examinations indicated that the three lesions were all pituitary adenomas with atypical cellular proliferation. The results of the immunohistochemical examinations were ACTH (+), hGH (low +), PRL (low +) and Ki67 (+;